ABSTRACT
The study reported here presents a detailed description of what it is like to parent a child with juvenile Huntington's disease in families across four European countries. Its primary aim was to develop and extend findings from a previous UK study. The study recruited parents from four European countries: Holland, Italy, Poland and Sweden,. A secondary aim was to see the extent to which the findings from the UK study were repeated across Europe and the degree of commonality or divergence across the different countries. Fourteen parents who were the primary caregiver took part in a semistructured interview. These were analyzed using an established qualitative methodology, interpretative phenomenological analysis. Five analytic themes were derived from the analysis: the early signs of something wrong; parental understanding of juvenile Huntington's disease; living with the disease; other people's knowledge and understanding; and need for support. These are discussed in light of the considerable convergence between the experiences of families in the United Kingdom and elsewhere in Europe.
Subject(s)
Huntington Disease/psychology , Parents/psychology , Adolescent , Adult , Child , Europe , Female , Humans , MaleABSTRACT
BACKGROUND: Existing research suggests that family caregivers of persons with Huntington's disease (HD) face a distinct series of problems, linked to the complex nature of the disease. Aubeeluck and Buchanan (Clin Genet, 71(5):434-445, 2007) developed and validated a disease-specific measure used to explore caregivers quality of life and assess the efficacy of therapeutic interventions. This current study builds on this research through the validation of French and Italian translations of the Huntington's disease quality of life battery for carers (HDQoL-C). METHOD: A total of 301 family carers completed the HDQoL-C. Participants were recruited through the "Euro-HDB" study which is measuring the burden in HD across Europe and the USA. RESULTS: Factor analysis demonstrated good internal consistency, reliability and congruent validity. Carers who cared for patients with less clinically severe symptoms reported significantly better QoL than carers of patients with more clinically severe symptoms. DISCUSSION: Findings indicate the HDQoL-C is multi-lingual, multi-cultural and easily applicable in other languages.
Subject(s)
Caregivers/psychology , Huntington Disease/psychology , Quality of Life/psychology , Surveys and Questionnaires/standards , Adult , Aged , Factor Analysis, Statistical , Family , Female , France , Humans , Italy , Male , Middle Aged , Psychometrics/statistics & numerical data , Reproducibility of Results , Severity of Illness Index , Socioeconomic Factors , TranslationsABSTRACT
Health-related quality-of-life instruments are critical for assessing disease burdens. Generic tools allow comparison between diseases but do not discriminate between disease severities. Specific tools also tend to be more sensitive. No specific tool is available to assess quality of life in patients with Huntington's disease (HD). In the context of the European study on HD burden, a specific tool was created: the Huntington Quality of Life Instrument (H-QoL-I). The aim of this study was to optimize the content and validate the H-QoL-I. After a semistructured interview with patients, caregivers and HD specialists, we conducted a patient focus group. A self-reported questionnaire was then developed in French and Italian. A total of 252 patients were recruited to answer the questionnaire. Face, internal and external validities were examined using a variety of methods. The shortened H-QoL-I that resulted from the successive analyses comprises 11 items, which are divided into three dimensions: motor functioning (four items), psychology (four items) and socializing (three items). These three domains were identified as being essential to cover the full domain of the quality of life for patients affected by HD. The H-QoL-I showed an acceptable reliability (Cronbach's α>0.84). Factor analyses demonstrated satisfactory construct validity. Moreover, the item internal consistency and item discriminant validity criteria were fulfilled. No differential item functioning was detected. External validity supported the scale's robustness. These data support the validity of the H-QoL-I in patients with HD.
